Počet záznamů: 1
ISL1 is necessary for auditory neuron development and contributes toward tonotopic organization
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SYSNO ASEP 0568863 Druh ASEP J - Článek v odborném periodiku Zařazení RIV J - Článek v odborném periodiku Poddruh J Článek ve WOS Název ISL1 is necessary for auditory neuron development and contributes toward tonotopic organization Tvůrce(i) Filova, Iva (BTO-N) ORCID
Pysaněnko, Kateryna (UEM-P) ORCID
Tavakoli, Mitra (BTO-N)
Vochyánová, Simona (BTO-N)
Dvořáková, Martina (BTO-N) ORCID
Bohuslavová, Romana (BTO-N) RID
Smolík, Ondřej (BTO-N)
Fabriciová, Valeria (BTO-N)
Hrabalová, Petra (BTO-N) RID
Benešová, Šárka (BTO-N)
Valihrach, Lukáš (BTO-N) RID, ORCID
Černý, Jiří (UMG-J)
Yamoah, E. N. (US)
Syka, Josef (UEM-P) RID
Fritzsch, B. (US)
Pavlínková, Gabriela (BTO-N) RID, ORCIDCelkový počet autorů 16 Číslo článku e2207433119 Zdroj.dok. Proceedings of the National Academy of Sciences of the United States of America. - : National Academy of Sciences - ISSN 0027-8424
Roč. 119, č. 37 (2022)Poč.str. 12 s. Jazyk dok. eng - angličtina Země vyd. US - Spojené státy americké Klíč. slova spiral ganglion neurons ; auditory nuclei ; inferior colliculus ; auditory maps ; auditory behavior Vědní obor RIV FH - Neurologie, neurochirurgie, neurovědy Obor OECD Neurosciences (including psychophysiology CEP GA20-06927S GA ČR - Grantová agentura ČR LM2018129 GA MŠMT - Ministerstvo školství, mládeže a tělovýchovy EF18_046/0016045 GA MŠMT - Ministerstvo školství, mládeže a tělovýchovy EF18_046/0015861 GA MŠMT - Ministerstvo školství, mládeže a tělovýchovy LM2018126 GA MŠMT - Ministerstvo školství, mládeže a tělovýchovy Způsob publikování Open access Institucionální podpora BTO-N - RVO:86652036 ; UEM-P - RVO:68378041 ; UMG-J - RVO:68378050 UT WOS 000895561900005 EID SCOPUS 85139290572 DOI 10.1073/pnas.2207433119 Anotace A cardinal feature of the auditory pathway is frequency selectivity, represented in a tono-topic map from the cochlea to the cortex. The molecular determinants of the auditory frequency map are unknown. Here, we discovered that the transcription factor ISL1 reg-ulates the molecular and cellular features of auditory neurons, including the formation of the spiral ganglion and peripheral and central processes that shape the tonotopic rep-resentation of the auditory map. We selectively knocked out Isl1 in auditory neurons using Neurod1Cre strategies. In the absence of Isl1, spiral ganglion neurons migrate into the central cochlea and beyond, and the cochlear wiring is profoundly reduced and dis-rupted. The central axons of Isl1 mutants lose their topographic projections and segrega-tion at the cochlear nucleus. Transcriptome analysis of spiral ganglion neurons shows that Isl1 regulates neurogenesis, axonogenesis, migration, neurotransmission-related machinery, and synaptic communication patterns. We show that peripheral disorganiza-tion in the cochlea affects the physiological properties of hearing in the midbrain and auditory behavior. Surprisingly, auditory processing features are preserved despite the significant hearing impairment, revealing central auditory pathway resilience and plastic-ity in Isl1 mutant mice. Mutant mice have a reduced acoustic startle reflex, altered prepulse inhibition, and characteristics of compensatory neural hyperactivity centrally. Our findings show that ISL1 is one of the obligatory factors required to sculpt auditory struc-tural and functional tonotopic maps. Still, upon Isl1 deletion, the ensuing central plastic-ity of the auditory pathway does not suffice to overcome developmentally induced peripheral dysfunction of the cochlea. Pracoviště Biotechnologický ústav Kontakt Monika Kopřivová, Monika.Koprivova@ibt.cas.cz, Tel.: 325 873 700 Rok sběru 2023 Elektronická adresa https://www.pnas.org/doi/full/10.1073/pnas.2207433119
Počet záznamů: 1